Cited by 109Genetics and Neurodevelopmental DisordersNeuroscience and Neuropharmacology ResearchMemory and Neural Mechanisms

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Conserved hippocampal cellular pathophysiology but distinct behavioural deficits in a new rat model of FXS

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Till, Sally M., Asiminas, Antonis, Jackson, Adam D., Katsanevaki, Danai, Barnes, Stephanie A., Osterweil, Emily K., Bear, Mark F., Chattarji, Sumantra, Wood, Emma R., Wyllie, David J.A., Kind, Peter C.·2015·Human Molecular Genetics

DOI 10.1093/hmg/ddv299PMC PMC4599667

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Abstract

Recent advances in techniques for manipulating genomes have allowed the generation of transgenic animals other than mice. These new models enable cross-mammalian comparison of neurological disease from core cellular pathophysiology to circuit and behavioural endophenotypes. Moreover they will enable us to directly test whether common cellular dysfunction or behavioural outcomes of a genetic mutation are more conserved across species. Using a new rat model of Fragile X Syndrome, we report that Fmr1 knockout (KO) rats exhibit elevated basal protein synthesis and an increase in mGluR-dependent long-term depression in CA1 of the hippocampus that is independent of new protein synthesis. These defects in plasticity are accompanied by an increase in dendritic spine density selectively in apical dendrites and subtle changes in dendritic spine morphology of CA1 pyramidal neurons. Behaviourally, Fmr1 KO rats show deficits in hippocampal-dependent, but not hippocampal-independent, forms of associative recognition memory indicating that the loss of fragile X mental retardation protein (FMRP) causes defects in episodic-like memory. In contrast to previous reports from mice, Fmr1 KO rats show no deficits in spatial reference memory reversal learning. One-trial spatial learning in a delayed matching to place water maze task was also not affected by the loss of FMRP in rats. This is the first evidence for conservation across mammalian species of cellular and physiological hippocampal phenotypes associated with the loss of FMRP. Furthermore, while key cellular phenotypes are conserved they manifest in distinct behavioural dysfunction. Finally, our data reveal novel information about the selective role of FMRP in hippocampus-dependent associative memory.

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Authors: Till, S. M., Asiminas, A., Jackson, A. D., Katsanevaki, D., Barnes, S. A., Osterweil, E. K., Bear, M. F., Chattarji, S., Wood, E. R., Wyllie, D. J. A., & Kind, P. C.
Affiliations: University of Edinburgh (GB)
Institute for Stem Cell Biology and Regenerative Medicine (IN)
Howard Hughes Medical Institute (US)
Tata Institute of Fundamental Research (IN)
Corresponding author: Kind, Peter C.
ORCID: Till, Sally M.
Asiminas, Antonis
Jackson, Adam D.
Katsanevaki, Danai
Barnes, Stephanie A.
Osterweil, Emily K.
Bear, Mark F.
Chattarji, Sumantra
Wood, Emma R.
Wyllie, David J.A.
Kind, Peter C.
Venue: Human Molecular Genetics
Publisher: Oxford University Press (OUP)
Published: 2015-08-04
Volume: 24
Issue: 21
Pages: 5977-5984
ISSN: 0964-6906 (ISSN-L)
1460-2083
DOI: 10.1093/hmg/ddv299
PMC: PMC4599667
OpenAlex: W1893201598
Cited by: 109
References: 28
Citations by year: 2026: 6 · 2025: 8 · 2024: 7 · 2023: 3 · 2022: 16 · 2021: 9 · 2020: 16 · 2019: 9 · 2018: 13 · 2017: 11 · 2016: 10 · 2015: 1
FWCI: 8.02
Type: journal article
Language: en
Open access: hybrid
doi.org
Licence: cc-by-nc
Locations: Human Molecular Genetics (publishedVersion, cc-by-nc)
Europe PMC (PubMed Central) (publishedVersion, cc-by-nc)
PubMed Central (publishedVersion, cc-by-nc)
PubMed (publishedVersion)
Edinburgh Research Explorer (University of Edinburgh) (submittedVersion, cc-by-nc)
DSpace@MIT (Massachusetts Institute of Technology) (submittedVersion, cc-by-nc)
Oxford University Press eBooks
Funders: Wellcome Trust (104116, 104116/Z/14/Z)
Research Councils UK
University of Edinburgh
Medical Research Council (MR/K014137/1, MR/M006336/1)
Topics: Life Sciences › Biochemistry, Genetics and Molecular Biology › Genetics › Genetics and Neurodevelopmental Disorders
Life Sciences › Neuroscience › Cellular and Molecular Neuroscience › Neuroscience and Neuropharmacology Research
Life Sciences › Neuroscience › Cognitive Neuroscience › Memory and Neural Mechanisms
Keywords: fragile X syndrome, FMRP, hippocampus, CA1 pyramidal neurons, dendritic spines, protein synthesis, synaptic plasticity, associative recognition memory, episodic-like memory, spatial memory, cross-species conservation, transgenic rats
MeSH terms: Animals, Disease Models, Animal, Female, Fragile X Syndrome, Hippocampus, Male, Memory Disorders, Neuronal Plasticity, Species Specificity, Rats, Sprague-Dawley, Pyramidal Cells, Maze Learning, Rats, Fragile X Mental Retardation Protein, Fragile X Messenger Ribonucleoprotein 1, Gene Knockout Techniques

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Conserved hippocampal cellular pathophysiology but distinct behavioural deficits in a new rat model of FXS

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